Cases reported "Granuloma, Plasma Cell"

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1/18. Rapid recurrence of an inflammatory myofibroblastic tumor in the right ventricular outflow tract.

    We describe an unusual case of rapid recurrence of a previously excised inflammatory myofibroblastic tumor of the right ventricular outflow tract in a 5-month-old infant. The infant is asymptomatic 18 months after the second surgery. The very rare cardiac involvement, and the early relapse of the inflammatory pseudotumor, to the best of our knowledge, is a unique combination. The inflammatory myofibroblastic tumor, as known as a pseudotumor or plasma cell granuloma is an uncommon reactive lesion with unknown aetiology. It is found most commonly in the lung and a number of visceral organs, such as the spleen, liver, ileum, salivary glands, urinary bladder, larynx or brain or in the retroperitoneum and lymph nodes. To our knowledge only 9 cases have been published of such tumor arising within the heart.
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2/18. Case report: Calcifying fibrous pseudotumour of the adrenal gland.

    Calcifying fibrous pseudotumour is a distinct pathological entity usually occurring in the soft tissue of the extremities, trunk, axilla, pleura, mediastinum and peritoneum. This report describes the hitherto unreported occurrence of this tumour of the adrenal gland in a 10-year-old girl whose imaging findings closely resembled a neuroblastoma. This entity is a potential pitfall in diagnosing adrenal neuroblastoma.
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3/18. Plasma cell granuloma of the pituitary gland. Case report.

    BACKGROUND: Plasma cell granuloma, one of the subtypes of inflammatory pseudotumor, is non-neoplastic proliferation of plasma cells and occurs extremely rarely in the intracranial space. We encountered a case of plasma cell granuloma in the pituitary gland, which initially presented with left oculomotor palsy. CASE DESCRIPTION: A 34-year-old female presented with left ptosis. Magnetic resonance image (MRI) demonstrated an intrasellar mass extending to the pituitary stalk with dural enhancement. The lesion was subtotally resected by a transsphenoidal approach. Histopathological findings showed that the resected mass consisted of mature plasma cells, and some cells had infiltrated the surrounding normal pituitary tissue. Immunohistochemical study demonstrated that these cells were positive for leukocyte common antigen, kappa and lambda light chains of IgG, indicating polyclonality of these plasma cells. CONCLUSIONS: We propose that, in the case of an intrasellar mass with unusual neurological symptoms including oculomotor palsy, inflammatory disease of the pituitary gland such as plasma cell granuloma should be included in the differential diagnosis.
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4/18. Inflammatory myofibroblastic tumour (inflammatory pseudotumour) of the breast. Clinicopathological and genetic analysis of a case with evidence for clonality.

    Inflammatory myofibroblastic tumours (IMTs) were initially considered to be benign reactive processes, but cases with an unfavourable outcome have been reported. Moreover, clonal genetic alterations have recently been published in some cases, suggesting that IMT may represent a malignant neoplastic entity. This paper reports a case of IMT that developed in the mammary gland, an unusual site. The histological picture was characterized by a proliferation of spindle cells with little cellular atypia and rare mitoses, associated with a polymorphous inflammatory infiltrate. Their immunophenotype, characterized by the expression of vimentin, smooth muscle actin, and cytokeratins, corresponded to that of myofibroblasts. cytogenetic analysis revealed the clonal nature of the lesion. The modal karyotype was 48, X, ins(2;X)(q34;p21.2p22.2), 7, del(9)(p23), 19. Including the present observation, a 9p deletion has now been found in three cases of IMT. These observations show that IMT may be a clonal neoplasm, even in sites different from deep soft tissues.
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5/18. Plasma cell granuloma of the thyroid with Hashimoto's thyroiditis: report of a rare case.

    As only eight cases have been previously reported in the literature, plasma cell granuloma of the thyroid gland is a rare entity. This condition can be confused with a benign or malignant neoplastic thyroid process. In this article, we describe a new case of plasma cell granuloma of the thyroid gland that occurred in a 46-year-old man who also had Hashimoto's thyroiditis. This case represents only the second documented instance of a plasma cell granuloma of the thyroid occurring in the setting of Hashimoto's thyroiditis. Moreover, it is only the second case of a plasma cell granuloma that has been reported in a male.
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6/18. Pseudotumour orbitae as the initial manifestation in Wegener's granulomatosis in a 7-year-old girl.

    Wegener's granulomatosis (WG) is a systemic granulomatous vasculitis that typically affects the upper airways, lungs and kidneys. This form of vasculitis is extremely rare in children, the most common form of paediatric systemic vasculitis being Henoch-Schonlein purpura (HSP). Children with systemic vasculitis look ill and have multiple constitutional complaints, often with prolonged fever. As in adults, WG in children is a multisystem disease. There are no precise statistical data concerning ocular involvement in Wegener's granulomatosis in children. Ocular disease occurs in 50%-60% adult patients with WG and may be an extension of the sinus inflammation or an orbital pseudotumour independent of the sinus disease (15%-50%); sometimes it presents as an ocular myositis. The most common presenting sign of orbital disease is ptosis and anterior displacement of the eye, pain, blurred vision, eyelid swelling and reduction of eye motility. Here we present the case of a 7-year-old girl with WG whose initial manifestation was pseudotumour orbitae. This patient was negative for an initial c-ANCA test. biopsy specimens obtained from the lacrimal gland and kidney demonstrated granulomatous vasculitis. Treatment with glucocorticoids and cyclophosphamide led to complete, long-lasting remission.
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7/18. Vanek's tumor (inflammatory fibroid polyp). Report of 18 cases and comparison with three cases of original Vanek's series.

    Eighteen cases of Vanek's tumors are presented. The patients included nine men and nine women between the ages of 45 and 93 years (mean, 66.2 years). Nine cases were clinically diagnosed as polyps of the gastric antrum, five cases as polyps of the stomach (not otherwise specified), one polyp was located in the ileum and the three remaining polyps in the small intestine (not otherwise specified). The thirteen polyps with available size information measured from 0.4 to 5 cm in the greatest diameter (mean, 2.2 cm). Immunohistochemically, the affections were positive for vimentin (18/18) and CD34 (15/18). All the cases negative for CD34 also lacked concentric onion skin-like formations of the spindle cells around glands and vessels. The different immunophenotype and absence of concentric formations could be explained by the existence of two different lesions commonly designated as Vanek's tumor (inflammatory fibroid polyp) or by the hypothesis of various evolutional stages. In the differential diagnosis, it is important to distinguish namely eosinophilic gastroenteritis, gastrointestinal stromal tumor, inflammatory pseudotumor, hemangioendothelioma, and hemangiopericytoma. In contrast to gastrointestinal stromal tumors, genetically no substitution, deletion, or insertion occurred in c-kit exon 11 in all analyzed samples. Likewise, no deletion or insertion in part of c-kit exon 9 was observed.
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8/18. Benign inflammatory pseudotumour of the parotid gland: a case report and literature review.

    We describe a rare case of benign inflammatory pseudotumour of the parotid gland. The literature concerning this condition is discussed.
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9/18. Inflammatory pseudotumor of the parotid gland: report of a case with fine needle aspiration cytology.

    BACKGROUND: Inflammatory pseudotumor is a rare lesion of the parotid gland. It usually presents as a mass lesion; thus, the clinical and radiologicfeatures often suggest malignancy. To the best of our knowledge, fine needle aspiration cytologic findings in parotid inflammatory pseudotumor have not been reported previously. CASE: A 59-year-old male presented with a palpable right parotid mass. Computed tomography revealed a mass measuring 2.5 cm in diameter. Fine needle aspiration cytology showed inflammatory cells, foamy histiocytes and groups of spindle-shaped cells without cytologic atypia. A diagnosis of inflammatory pseudotumor was suggested and was confirmed on histology. CONCLUSION: In the presence of a clinically evident mass in the parotid gland and fine needle aspiration cytologic features of inflammatory cells with sheets of spindle cells, the diagnosis of inflammatory pseudotumor should be suspected. The differential diagnosis of this unusual parotid gland lesion principally includes sialadenitis and myoepithelioma.
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10/18. Inflammatory myofibroblastic tumor of the parotid gland: case report and review of the literature.

    An inflammatory myofibroblastic tumor, previously known as an inflammatory pseudotumor, is an uncommon neoplasm. This tumor, which has characteristic morphological and immunohistochemical features, is mostly seen in the lung. Herein we present a rare case of an inflammatory myofibroblastic pseudotumor of the parotid gland as well as a review of the literature. The patient was a 66-year-old man with recurrent painful swelling of the parotid gland. A total parotidectomy with preservation of the facial nerve branches was performed. The patient showed no signs of recurrence > 3 years after surgery. The presence of clonal cytogenic abnormalities supported the neoplastic origin of this process. The treatment consisted of complete resection. Clinicians should however be aware that an inflammatory myofibroblastic tumor may mimic a reactive process.
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